Interactive Cardiovascular and Thoracic Surgery 3:118-120(2004)
© 2004 European Association of Cardio-Thoracic Surgery
Fatal post-operative gastro intestinal hemorrhage because of angio-dysplasia of small intestine in aortic regurgitation
Praveen Varmaa,*,
Manoranjan Misraa,
Vishnupuri Venkatraman Radhakrishnanb and
Kurur Sankaran Neelakandhana
a Division of Cardiovascular and Thoracic surgery, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India 695011
b Division of Pathology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India 695011
* Corresponding author. Tel.: +91-471-2444496; fax: +91-471-2446433
pkvarma{at}sctimst.ker.nic.in
Received July 28, 2003;
received in revised form September 8, 2003;
accepted September 15, 2003
 |
Abstract
|
|---|
Gastrointestinal bleeding due to angiodysplasia of the large intestine associated with calcific aortic stenosis is a well-known entity. Angiodysplasias are artero-venous malformations and they form one of the common causes of occult gastro-intestinal bleeding in the elderly. A 59-year-old man underwent aortic valve replacement for severe aortic regurgitation, developed severe gastro intestinal bleeding. Selective angiography was inconclusive. Exploratory laparotomy revealed angiodysplasia of the terminal ileum, which was resected. We report this case to draw attention to this rare cause of gastro intestinal bleeding and the difficulty in arriving at a diagnosis by the usual investigations.
Key Words: Aortic regurgitation; Angiography; Gastro-intestinal hemorrhage; Angiodyspalsia
|
1. Introduction
|
|---|
The association between unexplained gastro -intestinal (GI) bleeding and calcific aortic stenosis (AS) is a well-established entity (Heyde's syndrome). The bleeding is attributed to arterio-venous malformation (angiodysplasia) of the large intestine and it usually stops after aortic valve replacement. Angiodysplasia usually affects the large intestine, predominantly the caecum. It has also been reported in the small intestine and stomach. Small intestinal angiodysplasias are associated with occult GI bleed in elderly patients.
|
2. Case report
|
|---|
A 59-year-old male was admitted to our hospital with complaints of dyspnoea on exertion (NYHA CLASS III), exertional palpitations and easy fatigability. Physical examination revealed peripheral signs of severe aortic regurgitation (AR). Cardiovascular examination detected 3/6 early diastolic murmur in the aortic area. His hemoglobin (Hb) was 115 Gms/l with a hematocrit of 34%. X-Ray chest revealed cardiomegaly (cardio-thoracic ratio of 75%) with left ventricular (LV) apex. Electrocardiogram showed normal sinus rhythm with LV volume overload. Trans thoracic echocardiogram (TTE) showed severe aortic regurgitation and a dilated LV with ejection fraction of 74%. Coronary angiogram showed normal coronary anatomy and aortic root injection revealed severe AR. He underwent aortic valve replacement (AVR) with 23 mm TTK-Chitra heart® valve prosthesis (Tilting disc valve developed in our institute and marketed by TTK Pharma, Chennai, India). He was weaned off cardio pulmonary bypass easily without ionotropic support and extubated after 6 h. On the third post-operative day, he was restarted on enalapril [angiotensin converting enzyme (ACE) inhibitor]. He developed severe hypotension and circulatory collapse after 4 h and was re-admitted in the intensive care unit; reintubated and vasopressor drugs noradrenalin (0.1 µg/kg per min) and adrenaline (0.2 µg/kg per min) were infused to support the circulation. There was no evidence of hypovolumia; Hb and hematocrit were normal.
TTE showed good LV and prosthetic valve function and there was no evidence of cardiac tamponade. The patient was weaned off the ionotropic support and was extubated on the sixth post-operative day. On the evening of the sixth post-operative day, he developed melena, which was managed conservatively. On the subsequent days, he continued to have melena. Oesophago-duodenoscopy and colonoscopy were done, which did not show any pathology. The patient was taken for selective visceral angiogram on three occasions, but they did not reveal any bleeding source. On the 30th post-operative day, he developed massive lower GI bleed with features of shock. He was resuscitated and taken for emergency visceral angiogram. This showed vascular malformation of the left colic artery, which was embolized using gel form particles (Fig. 1). Immediately after the procedure, he developed another bout of severe GI bleed and circulatory collapse. Exploratory laparotomy was done and revealed multiple vascular malformations in the terminal ileum with a Meckel's diverticulum. The area involved was resected and an end-to-end anastomosis was carried out. Meckel's diverticulectomy was also performed. Histopathological examination confirmed the diagnosis of angiodysplasia of the small intestine (Fig. 2). Re-laparotomy performed after 4 days for severe abdominal distention showed complete anastomotic dehiscence. Anastomosis was re-fashioned but the patient expired due to septicemia and multi organ failure. Twenty-six units of blood were transfused during the period.
View larger version (168K):
[in this window]
[in a new window]
|
Fig. 1 Selective angiogram showing angiodysplasia of left colic artery, which was embolized with gel foam particles. The rest of the splanchnic vessels are poorly visualized because of vasospasm.
|
|
View larger version (129K):
[in this window]
[in a new window]
|
Fig. 2 Microphotograph showing dilated vascular channels (black arrow) in the submucosa of the intestine. The white arrow depicts the ulceration of the mucosa. Inset shows the lesions in high resolution.
|
|
|
3. Comment
|
|---|
Heyde [1] first described chronic gastrointestinal bleeding associated with calcific aortic stenosis in 1958. Boss and Rosenbaum described the pathology of bleeding in aortic stenosis in 1971 [2]. The bleeding was attributed to distention of mucosal and sub mucosal veins. These lesions were subsequently termed as angiodysplasia by Goldabini [3].
Angiodysplasias are arterio-venous malformations in the mucosa and sub mucosa of the gastrointestinal system. They occur most commonly in the caecum and right colon, but are also described in small intestine and stomach. Deficiency of high molecular weight multimers of von Willebrand factor associated with calcific AS causes chronic lower GI bleeding from these lesions After aortic valve replacement cessation of GI bleeding occurs due to recovery of high molecular weight multimers of von Willebrand factor [4].
However, angiodysplasia is one of the commonest causes of lower GI bleeding in elderly patients with reported incidence of 8–20%. The clinical presentation of angiodysplasia varies from an incidental finding in an otherwise asymptomatic person to occult bleeding or an acute massive hemorrhage [5]. The other important cause of lower GI bleed is diverticulosis, in this age group. Small bowel angiodysplasias accounts for 30–40% of cases of GI bleeding of obscure origin and represents the single most common cause for hemorrhage in this subset of patients [6]. Since more and more elderly patients are undergoing open-heart surgery in the present era, these lesions can form one of the important causes of lower GI bleed in the postoperative period.
Ikuta et al. [7] describes a patient who developed massive lower GI bleeding due to angiodysplasia of the small intestine following AVR in an elderly patient. The diagnosis was established by radioisotope-labeled red cell scintigraphy and selective angiography. The bleeding was controlled by embolization; however, the patient underwent emergency laparotomy and resection of the intestine due to gangrene of the bowel. They opine that the angiodysplasia may be related to aging rather than the aortic lesion per say as the bleeding occurred after AVR.
Those patients with major gastro intestinal bleeding following cardiac surgery should be investigated with oesophago-duodenoscopy, colonoscopy, scintigraphy and selective angiography. Colonoscopy and selective angiography can diagnose cause of bleeding, if the lesions are confined to colon, in the majority of the cases. Bleeding from the small intestine may be difficult to diagnose because of the organ's length and free intraperitoneal location. Although there are a variety of causes of small intestinal bleeding, angiodysplasias are the most common. The difficulty in localizing a bleeding site in the small bowel with sufficient accuracy to define a therapeutic target is well known. Several different tests can be used to identify the bleeding site preoperatively or intraoperatively [8], in spite of this, the lesion is likely to be missed in as much as 35% of cases [9]. Diagnostic laparotomy may be the only option in such cases.
Severe vaso-dilatation and circulatory collapse was probably related to ACE inhibitor. Vaso-constrictive agents administered for circulatory support could have caused splanchnic vessels vasoconstriction and mucosal ulceration causing these lesions to bleed. In this case, repeated selective angiography did not reveal the lesion because of severe vasospasm of splanchnic vessels (Fig. 1). Hence in such cases, early diagnostic laparotomy could be life saving. The construction of end-to-end anastomosis of the intestine was probably an unwise decision in such moribund patients, as the likelihood of anastomotic disruption is very high. Resection of the involved segment and the construction of temporary stoma would have been the more appropriate treatment.
doi:10.1016/S1569-9293(03)00233-0
|
References
|
|---|
- Heyde EC. Gastrointestinal bleeding in aortic stenosis. N Engl J Med. 1958;259:196
- Boss EG, Rosenbaum JM. Bleeding from the right colon associated with aortic stenosis. Am J Digest Dis. 1971;16:269–275[CrossRef][Medline]
- Baum S, Athana S, Waltman AS, Goldabini J, Shapiro RH, Warshaw AL, Ottinger LW. Angiodysplasia of the right colon, A cause of gastrointestinal bleeding. Am J Roentgenol 1977;129:789–94.
- Warkentin TE, Moore TC, Morgan DG. Gastrointestinal angiodysplasia and aortic stenosis. N Engl J Med. 2002;347(11):858–859[Free Full Text]
- Dodda G, Trotman BW. Gastrointestinal angiodysplasia. J Assoc Acad Minor Phys. 1997;8:16–19[Medline]
- Foutch PG. Angiodysplasia of the gastrointestinal tract: Am J Gastroenterol 1993:88807–18.
- Ikuta T, Shibata T, Hirai H, Dukui T, Suehiro S. Small intestinal bleeding from angiodysplasia after aortic regurgitation. J Heart Val Dis. 2003;12:458–460[Medline]
- Winskunas PF. Localization of bleeding site in the small bowel using a combined diagnostic approach. S D J Med. 1999;52:93–95[Medline]
- Al-Qahtani AR, Satin R, Stern J, Gordon PH. Investigative modalities for massive lower gastrointestinal bleeding. World J Surg. 2002;26:620–625[CrossRef][Medline]
This article has been cited by other articles:
|
|
|
|
 
P. K. Mishra, J. Kovac, J. de Caestecker, G. Fancourt, E. Logtens, and T. Spyt
Intestinal angiodysplasia and aortic valve stenosis: let's not close the book on this association
Eur. J. Cardiothorac. Surg.,
April 1, 2009;
35(4):
628 - 634.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
Top
Abstract
1. Introduction